The zebrafish motility mutant twitch once reveals new roles for rapsyn in synaptic function

Fumihito Ono, Anatoly Shcherbatko, Shin ichi Higashijima, Gail Mandel, Paul Brehm

Research output: Contribution to journalArticle

58 Citations (Scopus)

Abstract

Upon touch, twitch once zebrafish respond with one or two swimming strokes instead of typical full-blown escapes. This use-dependent fatigue is shown to be a consequence of a mutation in the tetratricopeptide domain of muscle rapsyn, inhibiting formation of subsynaptic acetylcholine receptor clusters. Physiological analysis indicates that reduced synaptic strength, attributable to loss of receptors, is augmented by a potent postsynaptic depression not seen at normal neuromuscular junctions. The synergism between these two physiological processes is causal to the use-dependent muscle fatigue. These findings offer insights into the physiological basis of human myasthenic syndrome and reveal the first demonstration of a role for rapsyn in regulating synaptic function.

Original languageEnglish (US)
Pages (from-to)6491-6498
Number of pages8
JournalJournal of Neuroscience
Volume22
Issue number15
StatePublished - Aug 1 2002
Externally publishedYes

Fingerprint

Zebrafish
Physiological Phenomena
Muscle Fatigue
Neuromuscular Junction
Muscle Weakness
Touch
Cholinergic Receptors
Fatigue
Stroke
Muscles
Mutation
peripheral membrane protein 43K

Keywords

  • Muscle fatigue
  • Myasthenia gravis
  • Rapsyn
  • Synapse development
  • Synaptic depression
  • Tetratricopeptide repeats

ASJC Scopus subject areas

  • Neuroscience(all)

Cite this

The zebrafish motility mutant twitch once reveals new roles for rapsyn in synaptic function. / Ono, Fumihito; Shcherbatko, Anatoly; Higashijima, Shin ichi; Mandel, Gail; Brehm, Paul.

In: Journal of Neuroscience, Vol. 22, No. 15, 01.08.2002, p. 6491-6498.

Research output: Contribution to journalArticle

Ono, Fumihito ; Shcherbatko, Anatoly ; Higashijima, Shin ichi ; Mandel, Gail ; Brehm, Paul. / The zebrafish motility mutant twitch once reveals new roles for rapsyn in synaptic function. In: Journal of Neuroscience. 2002 ; Vol. 22, No. 15. pp. 6491-6498.
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